Acquired cutaneous lymphangiectasia in two female patients with tuberculous lymphadenitis of the inguinal glands is described.

Keywords: Cutaneous lymphangiectasia, Tuberculous lymphadenitis

Introduction

Cutaneous lymphangiectasia represents a rare cutaneous condition resulting from lymphatic obstruction caused by a spectrum of scarring processes. We report 2 such cases that occurred as a consequence of lymph node tuberculosis.

Case 1

A 38- year -old woman was seen with elephantiasis and multiple warty papules of the vulva of 4 years duration. The lesions occasionally ruptured and got infected. Past medical history was significant for bilateral inguinal swellings of 8 years duration. The inguinal swellings used to rupture and healed gradually with scarring in a period of 6 months. She also had irregular fever. There was no personal or family history of pulmonary tuberculosis or any history of genital ulcer.

The pubic region was oedematous and the labia majora were swollen and firm in consistency and studded with multiple papulovesicles [Figure - 1]. Inguinal region showed a few non tender, matted lymph nodes of size 2x1.5cm and scars on both sides. Systemic examination was normal.

The lymph node biopsy was consistent with tuberculous lymphadenitis and the biopsy of a cutaneous lesion showed dilated lymph spaces lined with endothelium. Other investigations were normal except for positive Mantoux text and raised ESR.

Inspite of adequate antituberculosis treatment the skin lesions persisted and hence she was sent for plastic surgical repair.

Case 2

A 65-year-old woman was seen with swelling of the external genitalia of 1 year duration and multiple vulva) papules and papulovesicles of 3 months duration. She gave history of (R) inguinal swelling 5 years ago which was incised. This persisted as a discharging sinus for a long time and gradually healed with scarring. Inguinal biopsy report was tuberculous adenitis and was put on antituberculosis drugs, which she took only for 2 months. After a year she developed similar lesions on the (L) inguinal region which also suppurated and led to discharging sinuses and gradually healed. One year later she noticed elephantiasis of the vulva and gradual development of occasionally pruritic papulovesicles.

There was no history of pulmonary tuberculosis in her or in other family members. She denied any history of genital ulcer.

Skin biopsy of the warty lesion showed hyperkeratosis and acanthosis. Multiple dilated lymph spaces were seen in the upper dermis.

Discussion

Majority of the cutaneous lymphangiomas are congenital in origin. Occasionally they arise from lymphatic obstruction due to malignancies,' radiation therapy,' cutaneous trauma' or secondary to infectious diseases like lymph node tuberculosis,[4] recurrent erysipelas, filariasis and lymphogranuloma venereum. Heuvel et al[4] described a case of vulval lymphangiectasia secondary to lymph node tuberculosis, exactly similar to our patients. Lymphangiecatses of the chest wall resulting from scarring scrofuloderma has been reported by Leonardo et al.[5] Lymphatic obstruction leads to dilatation of lymph channels in the superficial and middermis or even in the deep dermis or subcutaneous tissues. The overlying epidermis may show varying degrees of hyperkeratosis, acanthosis and papillomatosis and may appear to enclose the ectatic lymphatic channels.[6]

Support group for parents, patients, children who suffer from all forms of lymphangiectasia. This condition is caused by dilation of the lymphatics. It can affect the intestinal tract, lungs and other critical body areas.

Department of Dermatology, College of Medicine, Chungnam National University, Daejon, Korea.

Abstract

We report the case of a 69-year-old man with a history of multiple erythematous bullae on both forearms, which had been present for about 1 month. The lesions appeared after several years of topical corticosteroid application and photoageing. A biopsy revealed lymphangiectasia with solar elastosis and increase in the ratio of elastic to collagen fibres in the dermis. We suggest that this patient's lymphangiectasia resulted from abnormal structure and function of the dermis due to photoageing and steroid-related atrophy.

A previously undescribed case of acquired cutaneous lymphangiectasias on the abdomen in a patient with cirrhotic ascites where peritoneal mesothelial cells refluxed in the skin is discussed. A 56-year-old man previously submitted to liver transplantation presented with vesiculobullous lesions on the developed as his cirrhotic ascites progressed. Histology showed dilated lymphatic channels in the upper dermis lined by a single, discontinuous layer of flattened, monomorphous endothelial cells with endoluminal papillary projections. In the deep reticular dermis, we observed irregular thin- often jagged-walled vascular channels lined by a single layer of bland endothelial cells, dissecting the collagen bundles. Vessels in the lumen were medium to large bizarre-shaped polygonal cells with abundant eosinophilic cytoplasm and hyperchromatic and irregular nuclei, arranged in small clusters or as solitary units, focally in close contact with the endothelial lining or free floating within vessel cavities. Immunohistochemistry indicated atypical intraluminal cells to be positive for calretinin, a specific marker for mesothelial cells. Pathophysiologic mechanisms and problems of differential diagnosis of this unique clinicopathologic entity are discussed.